title: Two unusual cases of intractable hyperthyroidism responsive to octreotide: Munchausen syndrome or not?
abstract: BACKGROUND
  The effective treatment for patients with resistant hyperthyroidism is difficult.
  
  
  METHODS
  In this case report with 4-year follow-up data, we present 2 unusual cases of hyperthyroidism that were unresponsive to almost all antithyroid treatments including total thyroidectomy, but both were controlled with octreotide.
  
  
  RESULTS
  Cases 1 and 2 were both middle-aged women. They presented thyrotoxicosis with a low serum concentration of TSH and thyroidal radioactive iodine uptake (RAIU). The underlying causes, such as thyroiditis, metastatic thyroid cancer and struma ovarii were explored. Iodine-induced hyperthyroidism, particularly factitious hyperthyroidism was highly suspected, but there was no direct evidence to establish these diagnoses. In spite of good compliance, their thyrotoxicosis could not be controlled with large doses of PTU or MMI. β-blocker, methylprednisolone, radio-iodine therapy and even thyroidectomy were all attempted and failed. Short-acting octreotide was first administered to case 1 and then to case 2. Thyroid function improved greatly within 3 days in both cases. The doses of octreotide were tapered down to twice a week with consistent efficacy. During the follow-up periods, case 1 required octreotide 0.1mg twice per week and case 2 is on thyroid replacement therapy due to hypothyroidism. The recurrences of hyperthyroidism in both cases were again rapidly controlled with the increased dose of octreotide in case 1 and re-started the usage of octreotide in case 2.
  
  
  CONCLUSIONS
  The etiology of thyrotoxicosis in these 2 cases is not clear. In the absence of struma ovarii or wide-spread follicular thyroid cancer, factitious hyperthyroidism due to Munchausen syndrome should be considered first. The efficacy of the off-label use of octreotide in hyperthyroidism was highly effective (only) in these 2 cases.
authors.affiliation: Department of Endocrine and Metabolic Diseases, Shanghai Rui-jin Hospital, Shanghai Jiao-tong University School of Medicine, China. liujianmin09@yahoo.com.cn
authors.forename: Jian-Min
authors.initials: JM
authors.lastname: Liu
authors.forename: Li-Qun
authors.initials: LQ
authors.lastname: Gu
authors.forename: Lin
authors.initials: L
authors.lastname: Zhao
authors.forename: Zheng-Yi
authors.initials: ZY
authors.lastname: Tang
authors.forename: Li-Hao
authors.initials: LH
authors.lastname: Sun
authors.forename: Jie
authors.initials: J
authors.lastname: Hong
authors.forename: Wei-Qing
authors.initials: WQ
authors.lastname: Wang
authors.forename: Bang-Yao
authors.initials: BY
authors.lastname: Luo
authors.forename: Yong-Ju
authors.initials: YJ
authors.lastname: Zhao
authors.forename: Man-Yin
authors.initials: MY
authors.lastname: Xu
authors.forename: Xi
authors.initials: X
authors.lastname: Chen
authors.forename: Xu-Feng
authors.initials: XF
authors.lastname: Jiang
authors.forename: Chen-Mo
authors.initials: CM
authors.lastname: Zhu
authors.forename: Xiao-Long
authors.initials: XL
authors.lastname: Jin
authors.forename: Hong-Zhuan
authors.initials: HZ
authors.lastname: Chen
authors.forename: Yu-Yan
authors.initials: YY
authors.lastname: Tan
authors.forename: Guang
authors.initials: G
authors.lastname: Ning
authors.forename: Jia-Lun
authors.initials: JL
authors.lastname: Chen
mesh_terms: D000328:Adult; D005260:Female; D006801:Humans; D006980:Hyperthyroidism / Q000150:complications* / Q000188:drug therapy* / Q000473:pathology / Q000503:physiopathology; D009110:Munchausen Syndrome / Q000150:complications*; D015282:Octreotide / Q000627:therapeutic use*; D013965:Thyroidectomy; D013997:Time Factors; D017211:Treatment Failure
chemical_list: D015282:Octreotide